Schistosomiasis is a chronic enteropathogenic disease caused by blood flukes of the genus Schistosoma. Coexistence of schistosomiasis with Crohn's disease is very rare. To the best of our knowledge, this association has been described in literature only once. A 20-year-old male patient with a past medical history of appendectomy and ileocecal Crohn's disease, presented with abdominal pain and vomiting. Ileocolonoscopy showed an ulcerated and congested appearance of the upper rectum and sigmoid. Computed tomography scan revealed a circumferential thickening of the terminal ileum with luminal stenosis. Histopathological examination of the biopsy specimens revealed a focally ulcerated colonic epithelium. The lamina propria was fibrous harbouring a polymorphic inflammatory infiltrate including lymphocytes and plasma cells organized in lymphoid follicles admixed with eosinophils and neutrophils. In the submucosa, there were two well-preserved schistosoma eggs surrounded by a thick shell with a barely visible terminal spine. The final pathological diagnosis was colonic schistosomiasis associated with Crohn's disease. The patient underwent an ileocecal resection for stenosis of the terminal ileum complicated with enterocutaneous fistula. The postoperative course was uneventful. A stool examination and serology tests were planned for this patient who was lost to follow-up.